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Otoferlin Depletion Results in Abnormal Synaptic Ribbons and Altered Intracellular Calcium Levels in Zebrafish
Aayushi Manchanda1, Paroma Chatterjee1, Josephine A Bonventre2
1Molecular and Cellular Biology Program, Oregon State University, Corvallis, Oregon, USA.
Scientific Reports
|October 5, 2019
Summary
Otoferlin protein is crucial for hearing and balance. Its depletion in zebrafish causes synaptic defects and abnormal gene expression, highlighting its conserved role in hair cell function.
Area of Science:
- Neuroscience
- Genetics
- Developmental Biology
Background:
- Otoferlin is vital for sensory hair cell synapse function.
- Otoferlin mutations cause deafness and vestibular deficits.
- Its role in synaptic vesicle dynamics across species is not fully understood.
Purpose of the Study:
- To investigate the conserved functions of otoferlin in zebrafish.
- To characterize the effects of otoferlin depletion on hair cell synapses.
Main Methods:
- Otoferlin depletion in zebrafish larvae.
- Analysis of synaptic vesicle recycling and synaptic ribbons.
- Measurement of intracellular calcium concentrations.
- Gene expression analysis of hair cell-specific genes.
Main Results:
- Otoferlin depletion caused synaptic vesicle recycling defects and abnormal synaptic ribbons.
- Elevated resting calcium levels were observed in hair cells.
- Abnormal expression of calcium-binding proteins and nogo-related proteins was detected.
- Exogenous otoferlin partially rescued gene expression defects.
Conclusions:
- Otoferlin is essential for synaptic vesicle recycling and maintaining resting calcium levels in zebrafish hair cells.
- Otoferlin influences synaptic ribbon structure and hair cell-specific gene transcription.
- These findings suggest conserved roles for otoferlin in vertebrate auditory and vestibular systems.
